Development of a Core Outcome Set for the Surgical Treatment of Sacrococcygeal Teratoma in Children

Sacrococcygeal teratoma (SCT) is a rare congenital tumor, usually identified during the neonatal or early infancy period. Prognosis after surgical resection is generally favourable, although urological and anorectal dysfunction remain important long-term sequelae. Outcome reporting in SCT studies is highly heterogeneous in terms of both selection and definition of outcomes, limiting comparability and impeding evidence synthesis. A targeted search of the COMET database identified no existing Core Outcome Set (COS) for the surgical treatment of SCT. The Core Outcome Set will be developed in accordance with COMET and COS-STAD guidelines. The development process includes a systematic review (PROSPERO: CRD4202561872), a Delphi study with stakeholders from both high and low resource settings, and a final consensus meeting.

Contributors

Marijke E.B. Kremer (MD, PhD)
Joep P.M. Derikx (MD, PhD)
Ernest L.W. van Heurn (MD, PhD), principal investigator

Department of Pediatric Surgery, Emma Children’s Hospital, Amsterdam University Medical Centers, University of Amsterdam, Amsterdam, The Netherlands

Further Study Information

Current Stage: Ongoing
Date: October 2024 - December 2025
Funding source(s): This study is supported by a KiKa pilot project grant awarded to Prof. Dr. E.L.W. van Heurn for the development of a Core Outcome Set for the surgical treatment of sacrococcygeal teratoma in children (Grant number: KiKa 440)


Health Area

Disease Category: Child health

Disease Name: Sacrococcygeal teratoma

Target Population

Age Range: 0 - 18

Sex: Either

Nature of Intervention: Surgery

Stakeholders Involved

- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Researchers

Study Type

- COS for clinical trials or clinical research
- COS for practice

Method(s)

- Consensus meeting
- Delphi process
- Systematic review

The Core Outcome Set for the surgical treatment of sacrococcygeal teratoma will be developed in three phases:
1. A systematic literature review will be conducted to identify all outcomes reported in studies concerning the surgical treatment of SCT in children.
2. Two parallel Delphi procedures (two or three rounds) will be conducted among stakeholders from high income and low and middle income countries, to prioritize key outcomes.
3. A consensus meeting will be held with international experts and patient representatives to ratify the final COS.
The development and reporting of the COS will adhere to the COS-STAD and COS-STAR methodological standards.

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